Literature DB >> 8733056

Cerebellar ataxia, areflexia, pes cavus, optic atrophy, and sensorineural hearing loss (CAPOS): a new syndrome.

P Nicolaides1, R E Appleton, A Fryer.   

Abstract

There are a large number of well recognised syndromes comprising cerebellar ataxia in association with other neurological features. We report three family members who presented with a relapsing, early onset cerebellar ataxia, associated with progressive optic atrophy and sensorineural deafness. All three patients have areflexia (in the absence of a peripheral neuropathy), a pes cavus deformity, and show varying degrees of severity. Extensive neurological investigations have been normal, and the aetiology and pathophysiology of this disorder remain unclear. This may represent a separate syndrome of early onset cerebellar ataxia with associated features ("cerebellar ataxia plus"), which is likely to either have an autosomal dominant or maternal mitochondrial pattern of inheritance. The recognition of this association under the acronym of CAPOS (cerebellar ataxia, areflexia, pes cavus, optic atrophy, and sensorineural deafness) may help in the delineation of a new syndrome.

Entities:  

Mesh:

Year:  1996        PMID: 8733056      PMCID: PMC1050615          DOI: 10.1136/jmg.33.5.419

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  11 in total

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Authors:  W B MATTHEWS
Journal:  J Neurol Neurosurg Psychiatry       Date:  1950-11       Impact factor: 10.154

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Journal:  Arch Neurol       Date:  1973-10

5.  Optic atrophy, neural deafness, and distal neurogenic amyotrophy; report of a family with two affected siblings.

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Journal:  J Neurol Neurosurg Psychiatry       Date:  1970-12       Impact factor: 10.154

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Authors:  J P Bouchard; C Roberge; N M van Gelder; A Barbeau
Journal:  Can J Neurol Sci       Date:  1984-11       Impact factor: 2.104

8.  X-linked ataxia, weakness, deafness, and loss of vision in early childhood with a fatal course.

Authors:  W F Arts; M C Loonen; R C Sengers; J L Slooff
Journal:  Ann Neurol       Date:  1993-05       Impact factor: 10.422

9.  Optic and cochleovestibular degenerations in the hereditary ataxias. I. Clinico-pathological and genetic aspects.

Authors:  L van Bogaert; L Martin
Journal:  Brain       Date:  1974-03       Impact factor: 13.501

10.  Familial periodic cerebellar ataxia without myokymia maps to a 19-cM region on 19p13.

Authors:  B T Teh; P Silburn; K Lindblad; R Betz; R Boyle; M Schalling; C Larsson
Journal:  Am J Hum Genet       Date:  1995-06       Impact factor: 11.025

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  18 in total

1.  [Hereditary optic atrophies].

Authors:  C M Poloschek; W A Lagrèze
Journal:  Ophthalmologe       Date:  2009-09       Impact factor: 1.059

2.  The CAPOS mutation in ATP1A3 alters Na/K-ATPase function and results in auditory neuropathy which has implications for management.

Authors:  Lisbeth Tranebjærg; Nicola Strenzke; Sture Lindholm; Nanna D Rendtorff; Hanne Poulsen; Himanshu Khandelia; Wojciech Kopec; Troels J Brünnich Lyngbye; Christian Hamel; Cecile Delettre; Beatrice Bocquet; Michael Bille; Hanne H Owen; Toke Bek; Hanne Jensen; Karen Østergaard; Claes Möller; Linda Luxon; Lucinda Carr; Louise Wilson; Kaukab Rajput; Tony Sirimanna; Katherine Harrop-Griffiths; Shamima Rahman; Barbara Vona; Julia Doll; Thomas Haaf; Oliver Bartsch; Hendrik Rosewich; Tobias Moser; Maria Bitner-Glindzicz
Journal:  Hum Genet       Date:  2018-01-05       Impact factor: 4.132

3.  Functional consequences of the CAPOS mutation E818K of Na+,K+-ATPase.

Authors:  Christian P Roenn; Melody Li; Vivien R Schack; Ian C Forster; Rikke Holm; Mads S Toustrup-Jensen; Jens P Andersen; Steven Petrou; Bente Vilsen
Journal:  J Biol Chem       Date:  2018-11-08       Impact factor: 5.157

Review 4.  Novel pregnancy-triggered episodes of CAPOS syndrome.

Authors:  Irene J Chang; Margaret P Adam; Suman Jayadev; Thomas D Bird; Niranjana Natarajan; Ian A Glass
Journal:  Am J Med Genet A       Date:  2017-11-01       Impact factor: 2.802

Review 5.  The expanding spectrum of neurological phenotypes in children with ATP1A3 mutations, Alternating Hemiplegia of Childhood, Rapid-onset Dystonia-Parkinsonism, CAPOS and beyond.

Authors:  Matthew T Sweney; Tara M Newcomb; Kathryn J Swoboda
Journal:  Pediatr Neurol       Date:  2014-10-13       Impact factor: 3.372

6.  Genome sequencing identifies a novel mutation in ATP1A3 in a family with dystonia in females only.

Authors:  Robert Wilcox; Ingrid Brænne; Norbert Brüggemann; Susen Winkler; Karin Wiegers; Lars Bertram; Tim Anderson; Katja Lohmann
Journal:  J Neurol       Date:  2014-10-31       Impact factor: 4.849

7.  Hypergonadotropic hypogonadism, progressive early-onset spinocerebellar ataxia, and late-onset sensorineural hearing loss: case report and literature review.

Authors:  E Sarikaya; Cg Ensert; Hc Gulerman
Journal:  Balkan J Med Genet       Date:  2011-12       Impact factor: 0.519

8.  A novel recurrent mutation in ATP1A3 causes CAPOS syndrome.

Authors:  Michelle K Demos; Clara Dm van Karnebeek; Colin Jd Ross; Shelin Adam; Yaoqing Shen; Shing Hei Zhan; Casper Shyr; Gabriella Horvath; Mohnish Suri; Alan Fryer; Steven Jm Jones; Jan M Friedman
Journal:  Orphanet J Rare Dis       Date:  2014-01-28       Impact factor: 4.123

9.  ATP1A3 mutations can cause progressive auditory neuropathy: a new gene of auditory synaptopathy.

Authors:  Kyu-Hee Han; Doo-Yi Oh; Seungmin Lee; Chung Lee; Jin Hee Han; Min Young Kim; Hye-Rim Park; Moo Kyun Park; Nayoung K D Kim; Jaekwang Lee; Eunyoung Yi; Jong-Min Kim; Jeong-Whun Kim; Jong-Hee Chae; Seung Ha Oh; Woong-Yang Park; Byung Yoon Choi
Journal:  Sci Rep       Date:  2017-11-28       Impact factor: 4.379

10.  ATP1A3 Mutation in Adult Rapid-Onset Ataxia.

Authors:  Kathleen J Sweadner; Camilo Toro; Christopher T Whitlow; Beverly M Snively; Jared F Cook; Laurie J Ozelius; Thomas C Markello; Allison Brashear
Journal:  PLoS One       Date:  2016-03-18       Impact factor: 3.240

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